ZC3H7B-BCOR high-grade endometrial stromal sarcoma may present as myoma nascens with cytoplasmic signet ring cell change

Virchows Arch. 2020 Apr;476(4):615-619. doi: 10.1007/s00428-020-02744-5. Epub 2020 Jan 14.

Abstract

We report on 51-year-old woman who presented with brown discharge and postcoital bleeding due to myoma nascens-like polypoid mass distending cervical canal. Histologically, the tumor consisted of high-grade spindle cell component with up to 15 mitotic figures per 10 HPF and also low-grade leiomyoma-like areas with focal myxoid change and so far undescribed cytoplasmic signet ring cell change. Immunohistochemically Desmin, actin, and h-caldesmon were negative. Conversely, BCOR positive expression was coupled with Cyclin D1 positivity and was antibody clone dependent. The molecular NGS and FISH study identified reciprocal fusion gene ZC3H7B-BCOR. In conclusion, these findings further support the idea of routine reflex molecular testing of uterine mesenchymal tumors with unusual clinical presentation or in case malignancy is suspected. Lastly, we suggest ZC3H7B-BCOR rearranged high-grade endometrial stromal sarcoma might be considered as a tumor suitable for BCL6-targeted treatment.

Keywords: BCOR protein; Endometrial stromal; Female; Fusion; Oncogene proteins; Proto-oncogene proteins; RNA-binding proteins; Repressor proteins; Sarcoma; Signet ring cell; ZC3H7B protein.

Publication types

  • Case Reports

MeSH terms

  • Biomarkers, Tumor / genetics
  • Carcinoma, Signet Ring Cell / diagnosis
  • Carcinoma, Signet Ring Cell / pathology*
  • Endometrial Neoplasms / genetics
  • Endometrial Neoplasms / pathology*
  • Female
  • Humans
  • Middle Aged
  • Proto-Oncogene Mas
  • Proto-Oncogene Proteins / genetics
  • Proto-Oncogene Proteins / metabolism*
  • RNA-Binding Proteins / metabolism*
  • Repressor Proteins / metabolism*
  • Sarcoma, Endometrial Stromal / genetics
  • Sarcoma, Endometrial Stromal / pathology*
  • Uterine Neoplasms / pathology

Substances

  • BCOR protein, human
  • Biomarkers, Tumor
  • MAS1 protein, human
  • Proto-Oncogene Mas
  • Proto-Oncogene Proteins
  • RNA-Binding Proteins
  • Repressor Proteins
  • ZC3H7B protein, human